PUBLICATION
Loss of egli-1, the Caenorhabditis elegans Orthologue of a Downstream Target of SMN, Leads to Abnormalities in Sensorimotor Integration
- Authors
- Da Silva, J.D., Oliveira, S., Pereira-Sousa, J., Teixeira-Castro, A., Costa, M.D., Maciel, P.
- ID
- ZDB-PUB-191206-8
- Date
- 2019
- Source
- Molecular neurobiology 57(3): 1553-1569 (Journal)
- Registered Authors
- Keywords
- C. elegans, Feeding, Neural circuits, SMA, Serotonin, egli-1
- MeSH Terms
-
- Disease Models, Animal
- Caenorhabditis elegans/genetics
- Caenorhabditis elegans Proteins/genetics
- Caenorhabditis elegans Proteins/metabolism*
- Muscular Atrophy, Spinal/genetics*
- Muscular Atrophy, Spinal/physiopathology
- Animals, Genetically Modified
- Animals
- Locomotion/genetics*
- Repressor Proteins/genetics*
- PubMed
- 31797327 Full text @ Mol. Neurobiol.
Citation
Da Silva, J.D., Oliveira, S., Pereira-Sousa, J., Teixeira-Castro, A., Costa, M.D., Maciel, P. (2019) Loss of egli-1, the Caenorhabditis elegans Orthologue of a Downstream Target of SMN, Leads to Abnormalities in Sensorimotor Integration. Molecular neurobiology. 57(3):1553-1569.
Abstract
The connectome of Caenorhabditis elegans has been extensively studied and fully mapped, allowing researchers to more confidently conclude on the impact of any change in neuronal circuits based on behavioral data. One of the more complex sensorimotor circuits in nematodes is the one that regulates the integration of feeding status with the subsequent behavioral responses that allow animals to adapt to environmental conditions. Here, we have characterized a Caenorhabditis elegans knockout model of the egli-1 gene (previously known as tag-175). This is an orthologue of the stasimon/tmem41b gene, a downstream target of SMN, the depleted protein in spinal muscular atrophy (SMA), which partially recapitulates the SMA phenotype in fly and zebrafish models when mutated. Surprisingly, egli-1 mutants reveal no deficits in motor function. Instead, they show functional impairment of a specific neuronal circuit, leading to defects in the integration of sensorial information related to food abundance, with consequences at the level of locomotion adaptation, egg laying, and the response to aversive chemical stimuli. This work has demonstrated for the first time the relevance of egli-1 in the nervous system, as well as revealed a function for this gene, which had remained elusive so far.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping